Osteopetrosis is a rare hereditary bone disorder that results in an increase in bone density due to gene mutations and osteoclastic dysfunction. This may lead to cranial nerve compression, bone fractures and osteomyelitis. Osteomyelitis of the maxilla is rare even in osteopetrosis patients. We report on a case of a 25-year-old male who presented with multiple episodes of osteomyelitis of the maxilla following dental extractions. The patient was initially managed with the incision and drainage of an acute infection, and intravenous amoxicillin-clavulanic acid. This was followed by the debridement of necrotic bony margins and packing with bismuth iodoform paraffin paste (BIPP) as well as long-term clindamycin. Once osteomyelitis was clear, the primary closure was achieved with a buccal advancement flap and supported by an acrylic obturator. Challenges in the management are highlighted, including preparing for a surgical intervention a patient with chronic low hemoglobin levels and a lack of healthy bony margins in the maxilla. The literature is also reviewed for similar cases.
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